Recurrent hypoglycemia in a young female with nesidioblastosis: A case report and review of literature

Table 1 Summary of published adult-onset nesidioblastosis case reports (2020-2025)

Ref.	Country	Case summary
Izumo et al[7], 2025	Japan	A 37-year-old woman presented with Whipple’s triad and fasting hyperinsulinemic hypoglycemia. Imaging negative for insulinoma. SACI test localized hypersecretion to gastroduodenal artery. Underwent pancreatoduodenectomy, with histology confirming nesidioblastosis. Recurrence at 1 year required remnant pancreatectomy, resulting in long-term symptom resolution
Chen et al[8], 2022	United States	An 83-year-old man presented with biochemical derangements suggestive of insulinoma. Preoperative cross-sectional imaging, Dotatate PET, and visceral angiogram all failed to definitively localize a neuroendocrine tumor. SACI showed a subtle increase in insulin secretion from the tail of the pancreas. Distal showing diffused islet hyperplasia consistent with a diagnosis of nesidioblastosis. Postoperative euglycaemia was achieved
Kim et al[9], 2022	United States	Patient presented with worsening hypoglycemic symptoms for 1 year prior to presentation that eventually progressed to hypoglycemic seizures. The onset of this hypoglycemia was 5 years after Roux-en-Y gastric bypass surgery. Extensive neurological and metabolic evaluation excluded other causes. Subtotal pancreatectomy confirmed nesidioblastosis; postoperative management included acarbose, a competitive reversible inhibitor of pancreatic α-amylase and intestinal brush border α-glucosidases which slows carbohydrate absorption
Tu et al[10], 2023	China	A 48-year-old male who suffered from repeated morning and fasting palpitations, sweating, and severe disturbance of consciousness for 5 years. His blood glucose was found to be as low as 1.79 mmol/L during an attack. However, abdominal computed tomography showed no abnormalities. Magnetic resonance imaging and endoscopic ultrasonography demonstrated a nodular mass in the head of the pancreas, combined with hyperinsulinemia and high serum C-peptide. The patient was diagnosed with insulinoma and underwent Beger surgery; however, the postoperative pathological results showed nesidioblastosis
Dieterle et al[11], 2023	Germany	A 23-year-old man with long-standing presyncope and exercise-induced hypoglycemia. Fasting test showed endogenous hyperinsulinism. 68Ga-DOTA-Exendin-4 PET/CT demonstrated diffuse pancreatic uptake. Required subtotal then total pancreatectomy due to recurrence. A subtotal pancreatectomy was performed, and the diagnosis of diffuse, adult-onset nesidioblastosis was established histopathologically. After nine months, the symptoms recurred, making complete pancreatectomy necessary. Postoperative laboratory evaluation exhibited no residual endogenous C-peptide production
Lisboa et al[12], 2025	Brazil	A 47-year-old male patient, diagnosed with Nesidioblastosis, in 2014, after frequent loss of consciousness and without association with fasting, was submitted to Frey's technique, after failure with other procedures. The method adopted proved to be effective for correcting the patient's endocrine problem, as well as without sequelae to the gastrointestinal tract
Said et al[13], 2025	Egypt	A PET/CT male patient presented with recurrent attacks of hypoglycemia, neuroglycopenic symptoms, and weight loss, prompting evaluation for causes of hyperinsulinemic hypoglycemia. Imaging (Gallium Dotatate PET/CT, EUS) identified a pancreatic tail lesion, prompting distal pancreatectomy. Histopathology examination confirmed nesidioblastosis. Post-surgery, transient diabetes developed but hypoglycemia recurred at 4 months. Octreotide treatment failed and diazoxid treatment successfully resolved symptoms
Abdul-Hafez et al[14], 2025	Palestine	A 55-year-old woman with a history of gastric sleeve and Roux-en-Y gastric bypass surgeries presented with a 15-year history of recurrent hypoglycemic episodes. Her symptoms persisted despite medical therapy with octreotide, acarbose, and nifedipine. Imaging ruled out insulinomas, raising suspicion of non-insulinoma pancreatogenous hypoglycemia syndrome. The patient underwent laparoscopic subtotal distal pancreatectomy. Histopathological examination confirmed nesidioblastosis, revealing irregular islet distribution and β-cell hypertrophy. Post-surgery, the patient achieved euglycemia without recurrence of hypoglycemic episodes during follow-up
Valentim et al[15], 2025	Brazil	A 42-year-old non-diabetic man with a history of coronary artery disease and systemic arterial hypertension had persistent hypoglycemia associated with high insulin levels. Total pancreatectomy and splenectomy performed after multidisciplinary decision-making; histopathology confirmed nesidioblastosis

SACI: Selective arterial calcium injection; PET: Positron emission tomography; CT: Computed tomography; EUS: Endoscopic ultrasound.

Table 2 Key differences between Nesidioblastosis, insulinoma and dumping syndrome

Feature	Nesidioblastosis	Insulinoma	Dumping syndrome
Pathophysiology	Diffuse or focal β-cell hyperplasia with inappropriate insulin secretion	Insulin-secreting pancreatic neuroendocrine tumor	Rapid gastric emptying causing exaggerated insulin response
Typical age	Neonates	Adults	Any age after gastric or bariatric surgery
Common risk factors	Post-bariatric surgery, idiopathic	MEN1, sporadic	Gastric surgery (RYGB, sleeve gastrectomy, gastrectomy)
Timing of hypoglycemia	Fasting and/or postprandial	Predominantly fasting	Postprandial (1-3 hours after meals)
Relationship to meals	Variable	Often relieved by eating	Occurs after meals, especially high-carbohydrate meals
Insulin during hypoglycemia	Inappropriately elevated	Inappropriately elevated	Elevated postprandially only
C-peptide	Elevated	Elevated	Normal to mildly elevated
Selective arterial calcium stimulation test	Diffuse or regional insulin response	Focal insulin step-up	Negative
Histopathology	Diffuse or focal islet hyperplasia, β-cell hypertrophy	Well-circumscribed neuroendocrine tumor	Normal pancreatic histology
Treatment	Dietary modification, diazoxide, octreotide. Subtotal or total pancreatectomy in refractory cases	Surgical enucleation or resection	Dietary modification
Risk of recurrence	High (especially after partial resection)	Low after complete excision	Symptoms may persist but improve with diet
Postoperative diabetes risk	High	Low to moderate	None

RYGB: Roux-en-Y gastric bypass.